Potential value of FDG PET-CT in diagnosis and follow-up of TAFRO syndrome

Behnia, Fatemeh; Elojeimy, Saeed; Matesan, Manuela; Fajgenbaum, David C

doi:10.1007/s00277-016-2875-8

Cited by 9 publications

(12 citation statements)

References 7 publications

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“…Interestingly, iMCD‐TAFRO patients demonstrate some features consistent with the treatment failure group in our study, such as normal IgG levels and hyaline vascular/hypervascular histopathology. On the other hand, TAFRO patients typically have elevated CRP, anaemia and low albumin, and siltuximab has been reported to be effective in some iMCD‐TAFRO cases (Hawkins & Pillai, ; Behnia et al , ). Future studies including patients with iMCD‐TAFRO are needed to determine if the proposed predictive model would apply.…”

Section: Discussionmentioning

confidence: 99%

Predictors of response to anti‐IL6 monoclonal antibody therapy (siltuximab) in idiopathic multicentric Castleman disease: secondary analyses of phase II clinical trial data

et al. 2018

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Siltuximab is the only US Food and Drug Administration-approved treatment for idiopathic multicentric Castleman disease (iMCD), a rare haematological disorder associated with substantial morbidity and mortality. Although siltuximab induces a response in a significant proportion of iMCD patients via interleukin 6 (IL6) neutralization, it is not universally effective. To develop a predictive model of response, we performed an in-depth analysis of 38 baseline laboratory parameters in iMCD patients from the phase II siltuximab trial who met criteria for treatment response or treatment failure. Univariate analyses identified eight baseline laboratory parameters that were significantly different between responders and treatment failures: albumin, immunoglobulin G (IgG), immunoglobulin A, C reactive protein (CRP), fibrinogen, haemoglobin, sodium and triglycerides. Stepwise logistic regression analysis of these candidate parameters identified a top performing model that included fibrinogen, IgG, haemoglobin and CRP. Based on cross-validation of the final multivariate logistic regression model, the model accurately discriminated responders from those who failed treatment (area under the receiver operator characteristic curve 0·86, 95% confidence interval: 0·73-0·95). All four laboratory parameters associated with response to siltuximab have biological relationships with IL6 and acute inflammation. Our model suggests that iMCD patients with laboratory evidence of an inflammatory syndrome are the best candidates for siltuximab therapy.

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Section: Discussionmentioning

confidence: 99%

Predictors of response to anti‐IL6 monoclonal antibody therapy (siltuximab) in idiopathic multicentric Castleman disease: secondary analyses of phase II clinical trial data

et al. 2018

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“…Nakamura et al reported diffuse hypointensity of the bone marrow in T1-and T2-weighted magnetic resonance images in patients with TAFRO syndrome, and they assumed that these findings reflected a reduction in the fatty component corresponding to reconversion into hematopoietic marrow owing to anemia and thrombocytopenia [28]. Behnia et al mentioned the efficacy of FDG-PET for selecting a lymph node for biopsy, for ruling out malignancies, and for evaluating treatment response [29]. However, in the present case, biopsy of the right cervical lymph node, which did not show high uptake of FDG, was sufficient for pathological evaluation.…”

Section: Discussionmentioning

confidence: 99%

Severe refractory TAFRO syndrome requiring continuous renal replacement therapy complicated with Trichosporon asahii infection in the lungs and myocardial infarction: an autopsy case report and literature review

et al. 2018

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Background: TAFRO (thrombocytopenia, anasarca, fever, reticulin myelofibrosis/renal failure, and organomegaly) syndrome is a systemic inflammatory disorder and unique clinicopathological variant of idiopathic multicentric Castleman disease that was proposed in Japan. Prompt diagnosis is critical because TAFRO syndrome is a progressive and life threating disease. Some cases are refractory to immunosuppressive treatments. Renal impairment is frequently observed in patients with TAFRO syndrome, and some severe cases require hemodialysis. Histological evaluation is important to understand the pathophysiology of TAFRO syndrome. However, systemic histopathological evaluation through autopsy in TAFRO syndrome has been rarely reported previously. Case presentation: A 46-year-old Japanese man with chief complaints of fever and abdominal distension was diagnosed with TAFRO syndrome through imaging studies, laboratory findings, and pathological findings on cervical lymph node and bone marrow biopsies. Interleukin (IL)-6 and vascular endothelial growth factor (VEGF) levels were remarkably elevated in both blood and ascites. Methylprednisolone (mPSL) pulse therapy was initiated on day 10, followed by combination therapy with PSL and cyclosporine A. However, the amount of ascites did not respond to the treatment. The patient became anuric, and continuous renal replacement therapy was initiated from day 50. However, the patient suddenly experienced cardiac arrest associated with myocardial infarction (MI) on the same day. Although the emergent percutaneous coronary intervention was successfully performed, the patient died on day 52, despite intensive care. Autopsy was performed to ascertain the cause of MI and to identify the histopathological characteristics of TAFRO syndrome.(Continued on next page)

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“…therapy (siltuximab or tocilizumab) and 9/13 (69%) responded to anti-IL-6 therapy±corticosteroids, according to the case report authors' assessments; three patients who responded to anti-IL-6 ± corticosteroids relapsed before the time of publication (Table 3). [17][18][19][20][21][22][23][24][25][26][27][28][29] We also assessed all of the iMCD-TAFRO and iMCD- Anti-IL-6 + rituximab or cyclosporine ± CS iMCD-TAFRO 5 5 3…”

Section: Real-world Datamentioning

confidence: 99%

Insufficient evidence exists to use histopathologic subtype to guide treatment of idiopathic multicentric Castleman disease

Fajgenbaum

Goodman

et al. 2020

American J Hematol

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Potential value of FDG PET-CT in diagnosis and follow-up of TAFRO syndrome

Cited by 9 publications

References 7 publications

Predictors of response to anti‐IL6 monoclonal antibody therapy (siltuximab) in idiopathic multicentric Castleman disease: secondary analyses of phase II clinical trial data

Predictors of response to anti‐IL6 monoclonal antibody therapy (siltuximab) in idiopathic multicentric Castleman disease: secondary analyses of phase II clinical trial data

Severe refractory TAFRO syndrome requiring continuous renal replacement therapy complicated with Trichosporon asahii infection in the lungs and myocardial infarction: an autopsy case report and literature review

Insufficient evidence exists to use histopathologic subtype to guide treatment of idiopathic multicentric Castleman disease

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